You are on page 1of 5

BRIEF REPORT

A Rare Case of Neck Pain: Acute Longus Colli


Calcific Tendinitis in a Possibly
Immunocompromised Individual
Kerlie Estimable, MD, Cynthia Rizk, MD, and George G. A. Pujalte, MD
We present a rare case of severe neck pain in a 45-year-old man with severe hidradenitis suppurativa
who was participating in a study involving adalimumab. The neck pain was associated with acute longus
colli calcific tendinitis, which is a noninfectious inflammatory response in the longus colli tendons secondary to deposition of calcium hydroxyapatite crystal. The diagnosis was made by computed tomography, which showed calcifications and deposits, and magnetic resonance imaging, which showed a retropharyngeal effusion. Ears, Nose, and Throat Services performed a fiberoptic scope examination, which
revealed a patent airway and no drainable abscess. Nonsteroidal anti-inflammatory drugs resulted in a
dramatic improvement in the patients clinical symptoms. In acute longus colli tendinitis, differentiating
retropharyngeal aseptic effusion from infection is important. Of note, the confounding factor in this
case was that the patient was blinded to whether he was receiving the placebo or adalimumab, so
whether the patient was immunosuppressed and at risk for infection was unknown. Clinician familiarity
and education concerning acute calcific longus colli tendinitis may lead to decreased costs stemming
from incorrect diagnosis and unnecessary treatment. (J Am Board Fam Med 2015;28:146 150.)
Keywords: Neck Pain, Tendinitis

Acute calcic tendinitis of the longus colli muscle is


a rare condition that causes severe neck pain. The
clinical presentation is commonly mistaken for
other life-threatening conditions, such as neck abscess, trauma, cervical disk herniation, and neoplasm.1 Symptoms are nonspecic and include
odynophagia, stiffness, neck pain, mild fever, and
limitation of cervical motion. Pertinent laboratory
values may include elevation of erythrocyte sedimentation rate, C-reactive protein, and white
blood cell count.1,2
Acute calcic tendinitis of the longus colli is
presumed to arise from a noninfectious inamma-

This article was externally peer reviewed.


Submitted 21 April 2014; revised 9 August 2014; accepted
13 August 2014.
From the Departments of Family and Community Medicine (KE, CR, GGAP) and Orthopaedics and Rehabilitation
(GGAP), Penn State Milton S. Hershey Medical Center,
Hershey, PA.
Funding: none.
Conict of interest: none declared.
Corresponding author: Kerlie Estimable, MD, Penn State
Hershey Family Medicine Residency, 4 Williamsburg Bldg,
Hershey, PA 17033 (E-mail: kestimable@yahoo.com).

146 JABFM JanuaryFebruary 2015

Vol. 28 No. 1

tory response of the superior oblique tendons of


the longus colli muscles secondary to deposition of
amorphous calcium hydroxyapatite crystal at the
anterior C1-C2 disk space; however, some cases
occur in more unusual areas (eg, C5-C6).2,3 Additional features seen on imaging include a nonsuppurative retropharyngeal effusion. Plain radiography is the rst line in diagnosis, but computed
tomography (CT) and/or magnetic resonance imaging (MRI) are the primary imaging methods used
for denitive diagnosis.3,4
We present a unique case of acute calcic longus
colli tendinitis in a patient, concurrently enrolled in
an adalimumab trial, who had incidental ndings of
rotatory subluxation of C3-C4. Both considerations delayed conrmation of the correct diagnosis.

Case Report
History
A 45-year-old male football coach presented with a
3-day history of abrupt onset of sharp posterior
cervical neck pain. He described it as severe pain

http://www.jabfm.org

Figure 1. Lateral view of the cervical plain radiograph


showed a large area of prevertebral soft-tissue
swelling (long arrows) from C1 to C5. Focal
calcification was noted inferior to the C1 arch (short
arrow).

was able to open his mouth only about 50%. He


had slight halitosis, but no oral lesions or palpable
adenopathy were noted. Cardiorespiratory, abdominal, musculoskeletal, and neurological examinations were all within normal limits. His white blood
cell count was mildly elevated at 12.85 K/L. Both
the erythrocyte sedimentation rate (49 mm/hour)
and C-reactive protein (7.06 mg/dL) also were elevated.

Emergency Department Course


Cervical spine radiographs showed prevertebral
soft-tissue swelling from C1 to C5 (Figure 1). CT
of the cervical spine revealed calcications anterior
to the C1-C2 level and a retropharyngeal softtissue collection extending from C2 to C4 (Figures
2 and 3). Orthopedic Surgery was consulted because a defect also was seen at C3. The orthopedist
felt that this was a congenital slight rotatory subluxation of C3-C4 (not visualized in the gures).
Ears, Nose, and Throat (ENT) Services were consulted and, using a beroptic scope, noted a patent
airway and no drainable abscess, determining that
the patient had a retropharyngeal phlegmon and
should be admitted for intravenous (IV) antibiotics.
He was admitted, and a neck MRI was ordered.
deep into his neck, radiating up to his temples,
and exacerbated by turning his head to the left. He
also had decreased neck range of motion, difculty
opening his jaw, and swallowing due to pain. With
each attempt to swallow, he felt a pulling sensation
and increased pain. He took ibuprofen and tramadol, which helped minimally. He reported 2 to 3
episodes of neck stiffness every year, but these were
usually self-limiting. His medical history was signicant for severe hidradenitis suppurativa, for
which he was participating in a study investigating
adalimumab as a treatment option compared with
placebo. He was blinded to the arm to which he was
assigned.

Figure 2. Sagittal computed tomography scan shows


an amorphous calcification (short arrow) anterior to
the C1C2 level. The disk space and heights were
maintained. A retropharyngeal soft-tissue fluid
collection extends from C2C4 (long arrows).

Physical Examination
The patients vitals were as follows: blood pressure,
140/100 mmHg; pulse rate, 100 beats/min; respiratory rate, 12 breaths/min; oxygen saturation,
98% on room air; and temperature, 36.8C. The
patient was sitting uncomfortably on the table with
a very stiff neck posture. Neck range of motion was
decreased; he had 10 degrees of exion, extension, and ear-to-shoulder movement bilaterally and

doi: 10.3122/jabfm.2015.01.140124

Acute Longus Colli Calcic Tendinitis

147

Figure 3. Computed tomography of the axial neck


shows a calcific density (arrow) at the superior
tendons of the longus colli at the C1C2 level.

Hospital Course
The differential diagnoses were cervical torticollis,
possible trauma, abscess, spontaneous retropharyngeal phlegmon, and deep neck infection related to
adverse effects of adalimumab. The working diagnosis was retropharyngeal phlegmon since no drainable
abscess was found, but the patient was given IV
ampicillin/sulbactam to address a possible deep
neck tissue infection given his involvement with the
adalimumab trial. Hydromorphone and cyclobenzaprine were given for pain control, with no significant improvement. MRI showed a focal, nonenhancing wall effusion dissecting bilaterally into the
longus colli muscles within the retropharyngeal
space, extending from C1 to C5 (Figures 4 and 5).
The diagnosis favored acute calcic longus colli
tendinitis, particularly because focal calcication of
the longus colli muscle at the C1-C2 level was
previously noted on the CT scan.
The Orthopedic Surgery service also reviewed
the MRI ndings and recommended IV steroids
and inpatient physical therapy. The patients adalimumab study supervisor was contacted; the primary investigator was on vacation, so whether the
patient was in the adalimumab or the placebo arm
of the study could not be determined.
A literature search for acute calcic longus colli
tendinitis was conducted, and few cases were found.
Some recommended nonsteroidal anti-inammatory drugs. Ketorolac (15 mg) given every 6 hours
drastically improved the patients condition by the
next day. He was discharged after 3 days with a

148 JABFM JanuaryFebruary 2015

Vol. 28 No. 1

Figure 4. Sagittal view of a T1-weighted magnetic


resonance image shows a bright retropharyngeal space
focal effusion (arrow) dissecting into the longus colli
muscles bilaterally from the anterior C1C5 level, the
absence of wall enhancement in the surrounding soft
tissue, and the absence of suppurative retropharyngeal
lymph nodes.

prescription to take diclofenac sodium 50 mg by


mouth 3 times a day as needed for 10 days. Ampicillin/sulbactam was switched to amoxicillin/clavulanate to be taken for 10 days in total.
Follow-up After Treatment
One week after treatment the patient was doing
well, able to tolerate a regular diet, and move his
neck without pain. Seven weeks later he continued
to be asymptomatic. He was still enrolled in the

Figure 5. Additional sagittal view shows a bright


retropharyngeal space focal effusion (arrow)
dissecting into the longus colli muscles bilaterally
from the anterior C1C5 level.

http://www.jabfm.org

Figure 6. T2-weighted magnetic resonance image shows


a bright retropharyngeal effusion (arrow).

adalimumab trial and he had completed the abovementioned treatment course. Repeat MRI showed
signicant improvement of the prevertebral, nonenhancing walled uid collection (Figure 7). Based
on a phone follow-up a couple of months later, the
patient had completed the adalimumab trial without recurrence of neck pain. It was conrmed that
patient has been received adalimumab and not the
placebo.

Discussion

4 6). The nal diagnosis then was conrmed as


acute calcic tendinitis rather than an infectious
process. This condition is also known as acute calcic prevertebral tendinitis or calcic retropharyngeal tendinitis.
The incidence of acute calcic longus colli tendinitis is still unknown because the diagnosis is
frequently missed.1,2,6 The disease occurs in both
sexes, with a slight female predominance. It normally occurs in the third to sixth decades of life.4,6
Since it was rst recognized, research about this
entity has depended primarily on case reports.6
The rst case reports were described in the
1960s.6,7 Later, in the 1990s, the calcications were
identied as amorphous hydroxyapatite deposits
within the superior tendons of the longus colli.8,9
Initial testing includes lateral neck radiography;
however, visualizing the calcication is difcult.10
CT of the cervical spine is the standard because it
is more sensitive for demonstration of calcium hydroxyapatite deposits.10 Compared with CT, MRI
is superior in delineating edema along the retropharyngeal space but is inferior in terms of demonstrating calcication.2,3,10 Retropharyngeal space
effusions have been reported in some case reports
similar to ours,2,4 but there has been no report in a

Figure 7. Sagittal view of a T1-weighted magnetic


resonance image shows interval resolution (arrow) of
the prevertebral enhancing fluid collection, with no
evidence of abscess or residual inflammation.

The uniqueness of this case stems from the fact that


our patient was at a potentially higher risk for
infection because of his involvement in a trial of
adalimumab for hidradenitis suppurativa. Adalimumab is a potent antagonist of tumor necrosis
factor biological activity.5 The US Food and Drug
Administration warns that adalimumab may put
patients at higher risk of serious infections.5 Because we did not know to which arm of treatment
the patient was assigned, the decision was made to
initiate antibiotics to address any possible infectious component to his condition.
In our patient acute calcic tendinitis of the
longus colli muscle was accompanied by a large
retropharyngeal space effusion and amorphous calcium deposit at the C1-C2 level of the cervical
spine. MRI revealed a nonenhancing wall effusion
within the retropharyngeal space extending from
C1-C5, without suppurative inammation (Figures

doi: 10.3122/jabfm.2015.01.140124

Acute Longus Colli Calcic Tendinitis

149

patient at high risk for disseminated infection,


thereby making an abscess a concurrent possibility.
There are 2 ways to differentiate a retropharyngeal space effusion from an abscess. First, there
must be no surrounding enhancement of the effusion. Second, suppurative inammatory retropharyngeal lymph nodes must be absent.3,4,8,10 Eastwood et al11 reported a similar description to
differentiate a retropharyngeal space effusion from
an abscess. Our patients MRI pointed toward an
effusion rather than an abscess.
Acute calcic tendinitis of the longus colli muscle is a self-limiting condition.2,3,6,9 12 The rst
line of treatment is nonsteroidal anti-inammatory
drugs.112 In severe, painful episodes patients may
also benet from corticosteroids.2,6 10 With the
right diagnostic imaging and proper treatment, initial improvement may occur within a couple of
days.2 Symptoms may take from 1 to 3 weeks to
completely resolve.1,2,4,6,9,10 Resolution of radiographic ndings may take weeks to months; however, follow-up imaging is not necessary.4,12

Conclusion
Awareness of the existence of acute calcic tendinitis
of the longus colli muscle may be helpful in differentiating this entity from conditions that cause similar
severe neck pain. An immunocompromised state may
make the diagnosis more difcult, given that abscesses
occur more frequently in such conditions. Adalimumab and other medications that may lead to an
immunocompromised state may also potentially put
patients at high risk for infection and abscess. Our
case emphasizes the importance of differentiating the
effusion of acute calcic tendinitis from a retropharyngeal abscess. Familiarity with imaging ndings
may prevent incorrect diagnosis and invasive surgical
procedures. Early recognition of the disease facilitates
the formation of an early treatment plan, which may
lead to rapid recovery and decreased costs.

150 JABFM JanuaryFebruary 2015

Vol. 28 No. 1

References
1. Jimenez S, Millan JM. Calcic retropharyngeal tendinitis: a frequently missed diagnosis. J Neurosurg
Spine 2007;6:77 80.
2. Zibis AH, Giannis D, Malizos KN. Acute calcic
tendinitis of the longus colli muscle: case report
and review of the literature. Eur Spine J 2013;22:
15.
3. Park SY, Jin W, Lee SH, Park JS, Yang DM, Ryn
KN. Acute retropharyngeal calcic tendinitis: a case
report with unusual location of calcication. Skeletal
Radiol 2010;39:81720.
4. Ellika SK, Payne SC, Patel SC, Jain R. Acute calcic
tendinitis of the longus colli: an imaging diagnosis.
Dentomaxillofac Radiol 2008;37:121 4.
5. American Society of Health System Pharmacists, Inc.
DynaMed. Adalimumab, record no. 232895. Ipswich
(MA): EBSCO Information Services; 1995; updated
September 7, 2011. Available from: http://search.
ebscohost.com/login.aspx?directtrue&siteDynaMed
&id232895. Accessed May 25, 2013.
6. Levy Z, Carroll J, Farley H. Benign nuchal rigidity:
the emergency department evaluation of acute prevertebral calcic tendonitis. West J Emerg Med
2012;13:114 6.
7. Vollmann R, Hammer G, Simbrunner J. Pathways in
the diagnosis of prevertebral tendinitis. Eur J Radiol
2012;81:114 7.
8. Ofah CE, Hall E. Acute calcic tendinitis of the
longus colli muscle: spectrum of CT appearances
and anatomical correlation. Br J Radiol 2009;82:
e11721.
9. Shin DE, Ahn CS, Choi JP. The acute calcic prevertebral tendinitis: report of two cases. Asian Spine
J 2010;4:1237.
10. Bladt O, Vanhoenacker P, Bevernage C, Oshoven
MV, Hoe LV, DHaenens P. Acute calcic prevertebral tendinitis. JBR-BTR 2008;91:158 9.
11. Eastwood JD, Hudgins PA, Malone D. Retropharyngeal effusion in acute calcic prevertebral tendinitis: diagnosis with CT and MR imaging. Am J
Neuroradiol 1998;19:1789 92.
12. Harnier S, Kuhn J, Harzheim A, Bewermeyer H,
Limmroth V. Retropharyngeal tendinitis: a rare differential diagnosis of severe headaches and neck
pain. J Head Face Pain 2008;48:158 61.

http://www.jabfm.org

You might also like